ZFIN ID: ZDB-FIG-170706-2
Colombo et al., 2015 - A zebrafish model of Poikiloderma with Neutropenia recapitulates the human syndrome hallmarks and traces back neutropenia to the myeloid progenitor. Scientific Reports   5:15814 Full text @ Sci. Rep.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Genes:
Fish:
Knockdown Reagent:
Anatomical Terms:
Stage: Long-pec
PHENOTYPE:
Fish:
Knockdown Reagents:
Observed In:
Stage Range: Long-pec to Day 5

Fig. 4

Overall phenotype of usb1 morphants.

Representative live-microscopy-4X magnification-pictures of 2 dpf (a) and 5 dpf (b) embryos injected with the same dosage (0.6 pmol/embryo) of Std (top), SMO-A (central) or SMO-B (bottom) morpholinos. The morphants display a smaller size, with defective and irregular pigmentation of the skin, as compared to the continuous stripes of the control embryos, and pericardial oedema. (c) Massive skeletal defects revealed by Alcian blue staining in usb1 morphants at 5 dpf. From left to right: diagram illustrating the normal pharyngeal arches-derived bone architecture19; ventral view of a control embryo (left panel) and representative pictures of the mild (left) and severe (right) phenotypes of SMO-A and SMO-B morphants. The Meckel’s (yellow in the colour-coded diagram), the palatoquadrate (green) and ceratohyal (purple) structures appear misshaped in both mildly- and severely-affected embryos; moreover, the ceratobranchial structures (light blue) appear disorganized or missing in half of SMO-A and in a third of SMO-B severely-affected embryos. (d) Reduction of gata1-positive cells (red fluorescence) (middle panels) and a regular vasculature pattern (green fluorescence) (lower panels) in 2 dpf tg(gata1:dsRED;flk1:GFP) embryos injected with SMO-A (0.7 pmol/embryo) (right) as compared to control (left). (e) Myeloid lineage defects evinced by a reduction in mpx-positive cells (green fluorescence) (lower panels) in 2 dpf tg(mpx:GFP;lyzC:dsRED) embryos injected with SMO-A (0.7 pmol/embryo) (right) as compared to control (left). Scale bars: (a–e) 200 μm.

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
DsRed s843Tg; sd2Tg control Long-pec nucleate erythrocyte IFL
s843Tg; sd2Tg + MO3-usb1 control Long-pec nucleate erythrocyte IFL
EGFP s843Tg; sd2Tg control Long-pec vasculature IFL
s843Tg; sd2Tg + MO3-usb1 control Long-pec vasculature IFL
GFP i114Tg; nz50Tg control Long-pec neutrophil IFL
i114Tg; nz50Tg + MO3-usb1 control Long-pec neutrophil IFL
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
AB + MO3-usb1 control Day 5 ceratohyal cartilage displaced, abnormal
Day 5 ceratohyal cartilage kinked, abnormal
Day 5 head decreased size, abnormal
Day 5 Meckel's cartilage misaligned with palatoquadrate cartilage, abnormal
Day 5 Meckel's cartilage shape, abnormal
Day 5 pericardium edematous, abnormal
Day 5 pharyngeal arch 3-7 aplastic/hypoplastic, abnormal
Day 5 pigmentation process quality, abnormal
Day 5 trunk edematous, abnormal
Day 5 whole organism decreased size, abnormal
Day 5 yolk edematous, abnormal
AB + MO3-usb1 standard conditions Long-pec head decreased size, abnormal
Long-pec pericardium edematous, abnormal
Long-pec pigmentation process quality, abnormal
AB + MO4-usb1 control Day 5 ceratohyal cartilage displaced, abnormal
Day 5 ceratohyal cartilage kinked, abnormal
Day 5 head decreased size, abnormal
Day 5 Meckel's cartilage misaligned with palatoquadrate cartilage, abnormal
Day 5 Meckel's cartilage shape, abnormal
Day 5 pericardium edematous, abnormal
Day 5 pharyngeal arch 3-7 aplastic/hypoplastic, abnormal
Day 5 pigmentation process quality, abnormal
Day 5 trunk edematous, abnormal
Day 5 whole organism decreased size, abnormal
Day 5 yolk edematous, abnormal
AB + MO4-usb1 standard conditions Long-pec head decreased size, abnormal
Long-pec pericardium edematous, abnormal
Long-pec pigmentation process quality, abnormal
Long-pec whole organism decreased size, abnormal
i114Tg; nz50Tg + MO3-usb1 control Long-pec neutrophil decreased amount, abnormal
Long-pec neutrophil GFP expression decreased amount, abnormal
Long-pec neutrophil GFP expression decreased distribution, abnormal
s843Tg; sd2Tg + MO3-usb1 control Long-pec nucleate erythrocyte decreased amount, abnormal
Acknowledgments:
ZFIN wishes to thank the journal Scientific Reports for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Sci. Rep.